nonstructural protein 1 of sars-cov-2 is a potent

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Journal Pre-proof Nonstructural protein 1 of SARS-CoV-2 is a potent pathogenicity factor redirecting host protein synthesis machinery toward viral RNA. Shuai Yuan, Lei Peng, Jonathan J. Park, Yingxia Hu, Swapnil C. Devarkar, Matthew B. Dong, Qi Shen, Shenping Wu, Sidi Chen, Ivan B. Lomakin, Yong Xiong PII: S1097-2765(20)30741-3 DOI: https://doi.org/10.1016/j.molcel.2020.10.034 Reference: MOLCEL 7696 To appear in: Molecular Cell Received Date: 11 August 2020 Revised Date: 5 October 2020 Accepted Date: 22 October 2020 Please cite this article as: Yuan, S., Peng, L., Park, J.J., Hu, Y., Devarkar, S.C., Dong, M.B., Shen, Q., Wu, S., Chen, S., Lomakin, I.B., Xiong, Y., Nonstructural protein 1 of SARS-CoV-2 is a potent pathogenicity factor redirecting host protein synthesis machinery toward viral RNA., Molecular Cell (2020), doi: https://doi.org/10.1016/j.molcel.2020.10.034. This is a PDF file of an article that has undergone enhancements after acceptance, such as the addition of a cover page and metadata, and formatting for readability, but it is not yet the definitive version of record. This version will undergo additional copyediting, typesetting and review before it is published in its final form, but we are providing this version to give early visibility of the article. Please note that, during the production process, errors may be discovered which could affect the content, and all legal disclaimers that apply to the journal pertain. © 2020 Published by Elsevier Inc.

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Page 1: Nonstructural protein 1 of SARS-CoV-2 is a potent

Journal Pre-proof

Nonstructural protein 1 of SARS-CoV-2 is a potent pathogenicity factor redirectinghost protein synthesis machinery toward viral RNA.

Shuai Yuan, Lei Peng, Jonathan J. Park, Yingxia Hu, Swapnil C. Devarkar, MatthewB. Dong, Qi Shen, Shenping Wu, Sidi Chen, Ivan B. Lomakin, Yong Xiong

PII: S1097-2765(20)30741-3

DOI: https://doi.org/10.1016/j.molcel.2020.10.034

Reference: MOLCEL 7696

To appear in: Molecular Cell

Received Date: 11 August 2020

Revised Date: 5 October 2020

Accepted Date: 22 October 2020

Please cite this article as: Yuan, S., Peng, L., Park, J.J., Hu, Y., Devarkar, S.C., Dong, M.B., Shen,Q., Wu, S., Chen, S., Lomakin, I.B., Xiong, Y., Nonstructural protein 1 of SARS-CoV-2 is a potentpathogenicity factor redirecting host protein synthesis machinery toward viral RNA., Molecular Cell(2020), doi: https://doi.org/10.1016/j.molcel.2020.10.034.

This is a PDF file of an article that has undergone enhancements after acceptance, such as the additionof a cover page and metadata, and formatting for readability, but it is not yet the definitive version ofrecord. This version will undergo additional copyediting, typesetting and review before it is publishedin its final form, but we are providing this version to give early visibility of the article. Please note that,during the production process, errors may be discovered which could affect the content, and all legaldisclaimers that apply to the journal pertain.

© 2020 Published by Elsevier Inc.

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Nonstructural protein 1 of SARS-CoV-2 is a potent pathogenicity factor 1

redirecting host protein synthesis machinery toward viral RNA. 2

3

Shuai Yuan1,6, Lei Peng2,4,6, Jonathan J. Park2,4, Yingxia Hu1, Swapnil C. Devarkar1, 4 Matthew B. Dong2,4, Qi Shen1, Shenping Wu5, Sidi Chen2,4*, Ivan B. Lomakin3* &Yong 5 Xiong1,7* 6 7 1 Department of Molecular Biophysics and Biochemistry, Yale University, New Haven, CT, 06511, 8 USA. 9 2 Department of Genetics, Yale University School of Medicine, New Haven, CT, 06520, USA. 10 3 Department of Dermatology, Yale university school of medicine, New Haven, CT, 06520, USA. 11 4 Systems Biology Institute, Yale University, West Haven, CT, 06516, USA. 12 5 Department of Pharmacology, Yale University, West Haven, CT, 06516, USA. 13 6 These authors contributed equally. 14 7 Lead contact. 15

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*Correspondence: [email protected] (S.C.), [email protected] (I.L.), 18

[email protected] (Y.X.) 19

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Summary 21

The causative virus of the COVID-19 pandemic, SARS-CoV-2, uses its nonstructural 22

protein 1 (Nsp1) to suppress cellular, but not viral, protein synthesis through yet 23

unknown mechanisms. We show here that among all viral proteins, Nsp1 has the 24

largest impact on host viability in the cells of human lung origin. Differential expression 25

analysis of mRNA-seq data revealed that Nsp1 broadly alters the cellular transcriptome. 26

Our cryo-EM structure of the Nsp1-40S ribosome complex shows that Nsp1 inhibits 27

translation by plugging the mRNA-entry channel of the 40S. We also determined the 28

structure of the 48S preinitiation complex formed by Nsp1, 40S, and the cricket 29

paralysis virus internal ribosome entry site (IRES) RNA, which shows that it is 30

nonfunctional due to the incorrect position of the mRNA 3’ region. Our results elucidate 31

the mechanism of host translation inhibition by SARS-CoV-2 and advances the 32

understanding of the impacts from a major pathogenicity factor of SARS-CoV-2. 33

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Introduction 35

SARS-CoV-2, which causes the worldwide COVID-19 pandemic affecting millions of 36

people, belongs to the β-coronaviruses (Coronaviridae Study Group of the International 37

Committee on Taxonomy of, 2020). The virus contains a positive-sense and single-38

stranded RNA that is composed of 5’-UTR, two large overlapping open reading frames 39

(ORF1a and ORF1b), structural and accessory protein genes, and 3’-poly-adenylated 40

tail (Lim et al., 2016). Upon entering the host cells, ORF1a and ORF1b are translated 41

and proteolytically processed by virus-encoded proteinases to produce functional 42

nonstructural proteins (Nsps) that play important roles in the viral infection and RNA 43

genome replication (Masters, 2006). Nsp1 is the first viral gene encoded by ORF1a 44

(Figure 1A) and is among the first proteins to be expressed after infection (Ziebuhr, 45

2005). It was shown that human SARS-CoV and group 2 bat coronavirus Nsp1 plays a 46

key role in suppressing the host gene expression (Kamitani et al., 2006; Narayanan et 47

al., 2008; Tohya et al., 2009). SARS-CoV Nsp1 has been shown to inhibit host gene 48

expression using a two-pronged strategy. Nsp1 targets the 40S ribosomal subunit to 49

stall the translation in multiple steps during initiation of translation and also induces an 50

endonucleolytic cleavage of host RNA to accelerate degradation (Kamitani et al., 2009; 51

Lokugamage et al., 2012). Nsp1 therefore has profound inhibitory effects on the host 52

protein production, including suppressing the innate immune system to facilitate the viral 53

replication (Narayanan et al., 2008) and potentially long-term cell viability 54

consequences. Intriguingly, viral mRNA overcomes this inhibition by a yet unknown 55

mechanism, likely mediated by the conserved 5’ UTR region of viral mRNA (Huang et 56

al., 2011; Tanaka et al., 2012). Taken together, Nsp1 acts as an important factor in viral 57

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lifecycle and immune evasion, and may be an important virulence factor causing the 58

myriad of long-term illnesses of COVID-19 patients. It has been proposed as a target for 59

live attenuated vaccine development (Wathelet et al., 2007; Zust et al., 2007). 60

It is common for RNA viruses to target the initiation step of the host protein 61

translation system to allow expression of the viral proteins (Jan et al., 2016). Most 62

cellular mRNAs have a 5’ 7-methylguanosine (m7G) cap structure, which is essential for 63

mRNA recruitment to the 43S preinitiation complex (PIC) through interaction with the 64

translation initiation factor (eIF) eIF4F. 43S PIC is formed by the 40S ribosomal subunit, 65

the ternary complex eIF2-GTP-Met-tRNAiMet, and the multi-subunit initiation factor eIF3. 66

Binding of the 43S PIC to the m7G-cap results in the loading of the mRNA in the mRNA-67

binding channel of the 40S to form the 48S PIC, and scanning of the mRNA from 5’ to 3’ 68

direction under control of eIF1A and eIF1, until the initiation codon AUG is placed in the 69

P site of the 40S. Base pairing of Met-tRNAiMet with AUG results in conformational 70

changes in the 48S PIC for joining the large 60S ribosomal subunit to form the 80S 71

ribosome primed for protein synthesis (Hinnebusch, 2014, 2017b; Hinnebusch et al., 72

2016). With the exception of type IV IRESes, such as the cricket paralysis virus (CrPV) 73

and Taura syndrome virus (TSV) IRESes, which do not require any host’s eIFs, all other 74

viruses may target different eIFs to redirect the host translational machinery on to their 75

own mRNA (Hertz and Thompson, 2011; Lozano and Martinez-Salas, 2015; Walsh and 76

Mohr, 2011). 77

We present here data demonstrating that among all viral proteins, Nsp1 causes 78

the most severe viability reduction in the cells of human lung origin. The introduction of 79

Nsp1 in human cells broadly alter the transcriptomes by repressing major gene clusters 80

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responsible for protein synthesis, mitochondria function, cell cycle and antigen 81

presentation, while inducing a broad range of factors implicated in transcriptional 82

regulation. We further determined the cryo-EM structures of the Nsp1-40S complex with 83

or without the CrPV IRES RNA, which reveal the mechanism by which Nsp1 inhibits 84

protein synthesis and regulates viral protein production. These results significantly 85

advance our understanding of the Nsp1-induced suppression of host gene expression, 86

the potential mechanisms of SARS-CoV-2 translation initiation, and the broad impact of 87

Nsp1 as a comorbidity-inducing factor. 88

89

Results 90

SARS-CoV-2 open reading frame (ORF) screen identifies Nsp1 as a major viral 91

factor that affects cellular viability 92

A recent study has mapped the interactome of viral protein to host cellular components 93

in human HEK293 cells (Gordon et al., 2020), suggesting that these viral proteins might 94

have diverse ways of interacting or interfering with the fundamental cellular machineries 95

of the host cell. We generated a non-viral over-expression vector (pVPSB) for 96

introduction of viral proteins into mammalian cells and testing their effect on cells 97

(Figure 1B). We first confirmed that the positive control GFP can be introduced into 98

virtually all cells at 100% efficiency, using flow cytometry analysis. We cloned 28 viral 99

proteins (27 of the 29 viral proteins and Nsp5 C145A mutation) as open reading frames 100

(ORFs) into this vector and introduce them into human cells by transfection. Intact 101

cDNAs of Nsp3 and Nsp16 had not been available when we performed the screen and 102

thus were not included in the screen, therefore the cellular phenotypes of these two viral 103

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proteins have not been tested here. We chose to first test H1299, an immortalized 104

cancer cell line of human lung origin. Although H1299 cells are not primary lung 105

epithelial cells, they have been utilized as a cellular model to study SARS-CoV, MERS 106

and SARS-CoV-2 (Hoffmann et al., 2020; Wong et al., 2015). 107

We introduced all 28 cloned ORFs individually in parallel to conduct a mini-108

screen of viral proteins’ effect on the viability of H1299 cells (Figures 1B and 1C). We 109

measured cell viability in two time points, 48 and 72 hours (h) post transfection. 110

Unexpectedly, we found Nsp1 as the sole “hit” with significant effect on cell viability at 111

both time points (Figure 1C). To validate the viability observations with increased 112

sensitivity, we generated an H1299 cell line with a constitutive firefly luciferase reporter 113

(H1299-PL), and confirmed that GFP can also be introduced into this cell line at near 114

100% efficiency (Figures S1A-C). We performed validation experiments, again with all 115

28 ORFs along with vector control, at 3 different time points (24, 48 and 72h). Across all 116

three time points, Nsp1-transfected H1299 cells have dramatically reduced luciferase 117

signal, an approximation of cell numbers (Figure 1D). We further repeat the same 118

experiments with the Vero E6 cell line, an African monkey (Cercopithecus Aethiops) 119

kidney derived cell line, commonly used in SARS-CoV-2 cellular studies (Blanco-Melo 120

et al., 2020; Hoffmann et al., 2020; Kim et al., 2020; Zhou et al., 2020). Consistently, we 121

observed a robust reduction of cellular viability in Vero E6 cells transfected with Nsp1 122

across all 3 time points (Figure S1D). These data revealed that among all SARS-CoV-2 123

proteins, Nsp1 has the largest detrimental effect on cell viability in H1299 and Vero E6 124

cells. 125

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Nsp1 mutants abolish cellular viability phenotype 127

To ensure that the observed reduction of cell viability is indeed from expression of 128

functional Nsp1, we tested three different mutants of Nsp1, including a truncation 129

mutation after residues 12 (N terminal mutant, N-trunc) and two double mutations that 130

have been reported to ablate the activity of SARS-CoV Nsp1 (Wathelet et al., 2007). As 131

SARS-CoV Nsp1 is highly homologous to SARS-CoV-2 Nsp1, we hypothesize that 132

these evolutionarily conserved amino acids may also have significant influence on the 133

activity of SARS-CoV-2 Nsp1. The point mutations include Nsp1 mutant3 that has 134

R124/K125 replaced with S124/E125 (R124S/K125E) and Nsp1 mutant4 that has N128/ 135

K129 replaced with S128/E129 (N128S/K129E). We performed cellular viability assays 136

with wild-type (WT) Nsp1 along with all three of its mutants. In both H1299-PL and Vero 137

E6-PL cells, we again observed that introduction of Nsp1 into cells significantly reduced 138

cell viability along 24, 48, and 72 hours post electroporation (Figures 1E and S1E). 139

Each of the three mutants (truncation, R124S/K125E and N128S/K129E) reverted this 140

phenotype to the vector control level, fully abolishing the cytotoxic effect of Nsp1 141

(Figures 1E and S1E). These results confirmed that functional Nsp1, but not its loss-of-142

function mutants, induce reduction of cellular viability when overexpressed in the two 143

mammalian cell lines. 144

We further tested if Nsp1 expression also leads to cell death. We introduced 145

Nsp1 into H1299 cells, along with controls of empty vector and several other viral 146

proteins (Nsp2, Nsp12, Nsp13, Nsp14, ORF9b, and Spike), and measured cellular 147

apoptosis at 48h post electroporation by flow cytometry analysis of cleaved Caspase 3 148

staining. We found that introduction of Nsp1, but not other viral proteins, induced 149

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apoptosis in H1299 cells (Figure S1G). To ensure the cellular apoptosis effect is indeed 150

from expression of functional Nsp1 protein, we performed the same apoptosis assay 151

with Nsp1 and the three non-functional mutants described above. Consistently, only 152

wild-type (WT) Nsp1 induced apoptosis in H1299-PL cells, whereas the three mutants 153

did not (Figure S1F). Replicates of this cleaved Caspase 3 flow assay with the 154

truncation mutation of Nsp1 confirmed that WT Nsp1, but not the loss-of-function 155

truncation mutant, induced apoptosis in H1299-PL cells (Figures 1F and 1G). 156

157

Transcriptome profiling of Nsp1-overexpressed cells 158

To unbiasedly investigate the global gene expression changes induced by Nsp1 or its 159

loss-of-function mutant form, we performed transcriptome profiling. We first confirmed 160

that Nsp1 is indeed over-expressed in host cells by qPCR using a custom-designed 161

NSP1-specific probe, at both 24 and 48 hours post electroporation (Figure 2A). We then 162

electroporated in quadruplicates for each of Nsp1, its truncation mutant, or vector 163

control plasmid into H1299-PL cells, and collected samples 24 hours post 164

electroporation for mRNA-seq. We collected 24h instead of 48h or 72h samples in order 165

to capture the earlier effect of Nsp1 on cellular transcriptome. We mapped the mRNA-166

seq reads to the human transcriptome and quantified the expression levels of annotated 167

human transcripts and genes (Table S3). Principle component analysis showed clear 168

grouping and separation of WT Nsp1, mutant Nsp1, or vector control groups (Figure 169

2B), confirming the overall quality of the Nsp1 mRNA-seq dataset. 170

Differential expression analysis revealed broad and potent gene expression 171

program changes induced by Nsp1 (Figure 2C; Table S3 and S4), with 5,394 genes 172

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significantly downregulated and 3,868 genes significantly upregulated (FDR adjusted q 173

value < 0.01). To examine the highly differentially expressed genes, we used a highly 174

stringent criteria (FDR adjusted q value < 1e-30), and identified 1,245 highly significantly 175

downregulated genes (top NSP1 repressed genes) and 464 highly significantly 176

upregulated genes (top Nsp1 induced genes) (Figure 2C; Table S3 and S4). In sharp 177

contrast, Nsp1 truncation mutant and the vector control showed no differential 178

expression in the transcriptome, even when using the least stringent criteria (FDR 179

adjusted q value < 0.05) (Figures S2A-B; Table S3 and S4). These data revealed that 180

Nsp1 alone can cause major alterations broadly in the transcriptome shortly (24h) after 181

its introduction into host cells, consistent with its cell viability phenotype (Figure 1). 182

183

Enriched pathway analysis on differentially expressed gene sets revealed strong 184

signatures of cellular transcriptome alterations by Nsp1 185

We globally examined the highly differentially expressed genes as a result of Nsp1 186

expression. To understand what these genes represent as a group, we performed 187

DAVID clustering and biological processes (BP) analysis on the 1,245 top Nsp1-188

repressed genes and the 464 top Nsp1-induced genes, respectively (Figure 2D; Table 189

S4). Enriched pathways in the top Nsp1-repressed genes showed that the most 190

significant gene ontology groups include functional annotation clusters of ribosomal 191

proteins and translation related processes, such as terms of ribonucleoprotein (RNP) 192

(Hypergeometric test, FDR-adjusted q = 6.30e-57), ribosomal RNA processing (q = 193

2.03e-28), and translation (q = 3.93e-28). Highly enriched Nsp1-repressed genes also 194

include the clusters of mitochondria function and metabolism (most terms with q < 1e-195

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15) and cell cycle and cell division (most terms with q < 1e-10), consistent with the 196

reduced cell viability phenotype. Other intriguing enriched Nsp1-repressed pathways 197

include ubiquitin/proteasome pathways and antigen-presentation activities, as well as 198

mRNA processing. We further performed gene set enrichment analysis (GSEA) that 199

takes into consideration both gene set and ranks of enrichment, and the results largely 200

validated the DAVID findings, with highly similar strongly enriched pathways (Figures 3A 201

and S2C). Analysis of highly differentially expressed genes between Nsp1 vs. Nsp1 202

mutant showed results virtually identical to those of Nsp1 vs. vector (Figures S2A-B, 203

Table S4). 204

We then examined the expression levels of the highly differentially expressed 205

genes in the context of enriched pathways in Nsp1, mutant Nsp1, or vector control 206

plasmid in H1299-PL cells. As shown in the heatmaps (Figure 3B), over 70 genes 207

involved in translation are strongly repressed upon introduction of Nsp1, including the 208

RPS, RPL, MRPS, MRPL family members, along with other translational regulators 209

such as AKT1. The repression effect on these genes is completely absent in the Nsp1 210

mutant group (Figure 3B). The strong repression effect also hit multiple members of the 211

gene families involved in mitochondria function, such as the COX, NUDFA, NUDFB and 212

NUDFS families (Figure 3C). Consistent with the cellular phenotypes, Nsp1 also 213

repressed a large number of mitotic cell cycle genes, including members in the CDK, 214

CDC and CCNB families, components of the centrosome, the anaphase promoting 215

complex and various kinases (Figure 3D). While part of the signal may be driven by 216

ribosomal and/or proteosomal genes, multiple genes involved in the mRNA processing 217

and/or nonsense-mediated decay nevertheless are significantly repressed by Nsp1 218

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(Figures S2D-E). Interestingly, DAVID BP enrichment analysis of Nsp1-repressed genes 219

also scored the antigen presentation pathway, mostly proteasome components along 220

with several MHC-I component members (Figure 3E). Concordantly, Nsp1-repressed 221

genes are also enriched in the ubiquitination and proteasome degradation pathways 222

(Figure S2F). 223

On the other hand, genes highly induced by Nsp1 hit a broad range of factors 224

implicated in transcriptional regulation, such as unfolded protein response regulators 225

(ATF4, XBP1), FOX family transcription factors (TFs) (FOXK2, FOXE1, FOXO1, 226

FOXO3), Zinc finger protein genes (ZFN217, ZFN567), KLF family members (KLF2, 227

KLF10), SOX family members (SOX2, SOX4), Homeobox genes (HOXD9, HOXC8, 228

HOXD13), GATA TFs (GATAD2B, GATA6), dead-box protein genes (DDX5, DHX36), 229

cell fate regulators (RUNX2, CREBRF, LIF, JUNB, ELK1, JAG1, SMAD7, BCL3, 230

EOMES); along with certain epigenetic regulators of gene expression such as the 231

SWI/SNF family members ARID1A, ARID1B, ARID3B, and ARID5B (Figure 3F). 232

Interestingly, highly upregulated genes are also slightly enriched in the MAPK/ERK 233

pathway, where Nsp1 expression induces multiple DUSP family members (Figure 3G). 234

The upregulated genes also include several KLF family members related to the process 235

of cellular response to peptide (Figure S2G). Again, the induction effect on these genes 236

is completely abolished in the Nsp1 mutant group (Figures 3F and 3G). These data 237

together showed that Nsp1 expression broadly and significantly altered multiple gene 238

expression programs in the host H1299-PL cells. 239

240

Cryo-EM structure reveals Nsp1 is poised to block host mRNA translation. 241

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To elucidate the mechanism of translation inhibition by Nsp1, we determined the cryo-242

EM structure of rabbit 40S ribosomal subunit complex with Nsp1 at 2.7 Å resolution 243

(Table 1, Figure S3). The density observed in the mRNA entry channel enabled us to 244

build an atomic model for the C-terminal domain of Nsp1 (C-Nsp1, amino acids (aa) 245

145-180) (Figure 4A). C-Nsp1 comprises two α-helices (α1, aa 154-160; α2, aa 166-246

179) and two short loops (aa 145-153 and 161-165), which blocks the mRNA entry 247

channel (Figure 4A-B). Besides the α-helices in the mRNA channel, extra globular 248

density between the ribosomal protein uS3 and rRNA helix h16 is observed at a lower 249

contour level, whose dimensions roughly matched the N-terminal domain of Nsp1 (aa: 250

13-127, N-Nsp1, PDB:2HSX) (Almeida et al., 2007) (Figure 4C). However, N-Nsp1 does 251

not appear to be stably bound to the 40S and the low local resolution of the cryo-EM 252

map in this region did not allow for an atomic model for the N-Nsp1. 253

C-Nsp1 bridges the head and body domains of the 40S ribosomal subunit 254

through extensive electrostatic and hydrophobic interactions with the ribosomal proteins 255

uS3 of the head, uS5 and eS30 and helix h18 of the 18S rRNA in the body (Figure 4D). 256

The negatively charged residues D152, E155 and E159 of C-Nsp1 interact with the 257

positively charged residues R117, R116, R143 and K148 of uS3, respectively (Figure 258

4E). In addition, K164 and H165 of Nsp1 inserts into the negatively charged pocket 259

formed by the backbone of U607, G625 and U630 of the rRNA h18. R171 and R175 of 260

C-Nsp1 interact with the negatively charged patch formed by G601, A604, G606 and 261

U607 of h18 (Figure 4E). Besides electrostatic contacts, a large hydrophobic patch of C-262

Nsp1, which is formed by F157, W161, L173 and L177, interacts with a complimentary 263

hydrophobic patch on uS5 formed by V106, I109, P111, T122, F124, V147 and I151 264

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(Figure 4E). Intriguingly, K164 and H165 of Nsp1, which have been shown to play an 265

important role in host translation inhibition, are conserved only in the 266

betacoronaviruses (beta-CoVs) (Figure S4). In addition, the other Nsp1 residues 267

interacting with the h18 of rRNA are also conserved only among the beta-CoVs (Figure 268

S4). This sequence conservation indicates that the hydrophobic interactions between C-269

Nsp1 and uS5 are likely universal in both alpha- and beta-CoVs, while the electrostatic 270

interactions between C-Nsp1 and the h18 of the 18S rRNA are conserved only in the 271

beta-CoVs. The extensive interactions result in C-Nsp1 plugging the mRNA entry 272

channel, which prevents the loading and accommodation of the mRNA (Figure 4B), 273

providing a structural basis for the inhibition of host protein synthesis by Nsp1 of SARS-274

CoV-2 and SARS-CoV reported previously (Kamitani et al., 2009; Kamitani et al., 2006). 275

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Nsp1 locks the 40S in a conformation incompatible with mRNA loading and 277

disrupts initiation factor binding 278

The ribosomal protein uS3 is conserved in all kingdoms. Together with h16, h18 and 279

h34 of 18S rRNA it constitutes the mRNA-binding channel and the mRNA entry site 280

(Graifer et al., 2014; Hinnebusch, 2017a). It has been shown that uS3 interacts with the 281

mRNA and regulates scanning-independent translation on a specific set of mRNAs 282

(Haimov et al., 2017; Sharifulin et al., 2015). Interestingly, conserved residues R116 and 283

R117 of uS3, which are crucial for stabilizing mRNA in the entry channel and 284

maintaining 48S PIC in the closed conformation, are interacting with D152, E155 of 285

Nsp1 in our structure (Dong et al., 2017; Hinnebusch, 2017a) (Figure 4E). Moreover, the 286

conformation of the 40S ribosomal subunit in Nsp1-40S complex is similar to that of 287

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‘closed state’ of 48S PIC with initiator tRNA locked in the P site and the latch closed 288

(Lomakin and Steitz, 2013), which is incapable of mRNA loading. The distance between 289

G610 (h18) and GLN179 (CA, uS3) is shortened from 19.4 Å in the ‘open state’ 48S PIC 290

(PDB:3JAQ) to 15.8 Å in Nsp1-40S ribosomal complex, which is similar to the distance 291

of 15.0 Å in the closed state 48S PIC (PDB:4KZZ) (Figure 4F). This shows that Nsp1 292

not only plugs the mRNA entry channel, but also keeps the 40S subunit in a 293

conformation that is incompatible with mRNA loading. 294

The known structure of the N-terminal domain of SARS-CoV (N-Nsp1) (Almeida 295

et al., 2007) (PDB ID: 2HSX) can be docked into the extra globular density between uS3 296

and rRNA helix h16 in the cryo-EM map (Figure 4G). This potential interaction between 297

N-Nsp1 and uS3 covers most of the uS3 surface on the solvent side, including the 298

GEKG loop of uS3 (aa: 60-63) that corresponds to the consensus GXXG loop 299

conserved in the KH domains of various RNA-binding proteins (Babaylova et al., 2019; 300

Graifer et al., 2014). Mutation of the GEKG loop to alanines does not abrogate the 301

ability of the 40S to bind mRNA and form 48S preinitiation complex (PIC). Instead, it 302

results in the formation of aberrant 48S PIC that cannot join the 60S ribosomal subunit 303

and assemble the 80S initiation complex (Graifer et al., 2014). Peculiarly, binding of 304

SARS-CoV Nsp1 to the ribosome led to the same effect (Kamitani et al., 2009). We 305

hypothesize that Nsp1 may prevent the formation of physiological conformation of the 306

48S PIC induced by uS3 interaction with translation initiation factors, such as the j 307

subunit (eIF3j) of the multi-subunit initiation factor eIF3 (Babaylova et al., 2019; Cate, 308

2017; Sharifulin et al., 2016). The eIF3 complex plays a central role in the formation of 309

the translation initiation complex (Cate, 2017; Hinnebusch, 2014). eIF3j alone binds to 310

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the 40S ribosomal subunit and stabilizes the interaction with eIF3 complex (Fraser et 311

al., 2004; Sokabe and Fraser, 2014). The binding site of eIF3j to 40S subunit is not 312

precisely determined. Cryo-EM and biochemical studies mapped it onto the mRNA 313

binding channel of the 40S, extending from the decoding center toward the mRNA entry 314

region, including the GEKG loop of uS3 (Aylett et al., 2015; Fraser et al., 2007; Hershey, 315

2015) (Figure 4G). 316

We tested if Nsp1 can compete with eIF3j for the binding to the 40S ribosomal 317

subunit. The result showed that Nsp1 indeed significantly reduces the binding between 318

eIF3j and the 40S (Figure 4H). The binding competition of eIF3j and Nsp1 to the 40S 319

was tested at different concentrations. There is little eIF3j binding to the 40S when the 320

concentration of eIF3j is equal or lower than that of Nsp1, and residual eIF3j binding 321

was observed only when its concentration is higher than that of Nsp1 (Figures 4H and 322

S5). By contrast, the binding of Nsp1 to the 40S is not affected even when eIF3j is in 323

excess. These results indicate that Nsp1 disrupts the binding of eIF3j to the 40S, 324

potentially by shielding the access to uS3 and the mRNA binding channel and/or by 325

making the conformation of the 40S unfavorable for eIF3j interaction. 326

327

Nsp1 prevents physiological conformation of the 48S PIC 328

It was shown previously that binding of SARS-CoV Nsp1 to the 40S ribosomal subunit 329

does not inhibit 48S PIC formation, but it suppresses 60S subunit joining (Kamitani et 330

al., 2009). To understand the effect of Nsp1 of SARS-CoV-2 on 48S PIC, we determined 331

a 3.3 Å resolution cryo-EM structure of Nsp1 bound to the 48S PIC assembled with the 332

cricket paralysis virus (CrPV) internal ribosome entry site (IRES) (Figures 5A and S6). 333

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CrPV IRES has become an important model for studies of the eukaryotic ribosome 334

during initiation, as it is able to directly recruit and assemble with 40S or 80S ribosome 335

without requiring any eIFs (Martinez-Salas et al., 2018). It was shown that SARS-CoV 336

Nsp1 inhibits translation of the CrPV IRES RNA (Kamitani et al., 2009). The use of 337

CrPV IRES allowed us to probe if Nsp1 completely inhibits mRNA binding to the 40S 338

subunit or it acts on the mRNA entry site only, as binding of the IRES may help fix 5’-339

region of the mRNA on the ribosome mRNA exit region, enabling the investigation of the 340

mRNA path on the 40S subunit in the presence of Nsp1. We first examined whether 341

Nsp1 affects binding of the IRES RNA to the 40S ribosomal subunit. The result shows 342

that Nsp1 and CrPV IRES can bind 40S ribosomal subunit simultaneously (Figure S6A-343

B). Consistently, both C-Nsp1 and the CrPV IRES can be seen in the cryo-EM map 344

(Figure 5A), where the Nsp1 C-terminal domain is inserted in the RNA entry channel in 345

the same way as in the Nsp1-40S complex without the IRES RNA (Figures 4A and 4B). 346

The local environment of C-Nsp1 in the ribosome RNA entry channel with or without the 347

IRES RNA is quite similar. No conformational changes were observed for C-Nsp1, 348

protein uS5 and rRNA h18, however, the head of the 40S subunit is moved by about 2.8 349

Å (Figure 5A) (discussed more below). 350

We fitted the high resolution structure of the CrPV IRES from the yeast 40S-CrPV 351

IRES complex(Murray et al., 2016) (PDB: 5IT9) into our cryo-EM map. Importantly, the 352

pseudoknot I (PKI) domain of the CrPV IRES, which is a structural mimic of the 353

canonical tRNA-mRNA interaction, is not seen in the cryo-EM map, suggesting that it is 354

dislodged from the 40S in the presence of Nsp1 (Figure 5B). Consistently, there would 355

be a clash between Nsp1 C-terminal domain and the 3’ region of the IRES RNA in the 356

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previously observed conformation bound to the 40S (Murray et al., 2016) (Figure 5B). 357

The conformation of the 40S head in the Nsp1-40S-CrPV IRES complex is different 358

from that in the Nsp1-40S complex (Figure 5C). The head in the Nsp1-40S-CrPV IRES 359

complex is in somewhat intermediate conformation compared to the Nsp1-40S and the 360

40S-CrPV IRES complexes (Figure 5C). This suggests that the Nsp1-40S interactions 361

resist the conformational changes induced by the IRES for translation initiation. 362

Conformational changes of the head domain of the 40S subunit play important role in 363

the mRNA loading and recruitment of the 60S subunit to form the 80S ribosome. Nsp1 364

limits the rotation of the head, which may have profound consequences interfering with 365

the joining of the 60S subunit and the formation of the 80S initiation complex. 366

367

Discussion 368

Viral infection is a complex process involving multiple components and certain viral 369

proteins are often in high abundance in cells during active viral replication (Astuti and 370

Ysrafil, 2020; Yoshimoto, 2020). Therefore, understanding the effects of each individual 371

viral protein on the cells provides important insights on the cellular impacts of viral 372

infection. Using a reductionist approach, we tested the gross cellular effect of 373

expressing most of the SARS-CoV-2 proteins individually, and found that among all 374

ORFs tested, Nsp1 showed the strongest deleterious effect on cell viability in H1299 375

cells of human lung epithelial origin. This is in concordance with previous observations 376

from related coronaviruses, such as mouse hepatitis virus (MHV) Nsp1 being a major 377

pathogenicity factor strongly reducing cellular gene expression (Zust et al., 2007), and 378

SARS-CoV Nsp1 inhibiting interferon (IFN)-dependent signaling and having significant 379

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effects on cell cycle (Wathelet et al., 2007). A recent study shows that SARS-CoV-2 380

Nsp1 shuts down mRNA translation in cells and suppresses innate immunity genes 381

such as IFNb and IL-8, although these experiments were conducted in HEK293T cells 382

of kidney origin, and only a small number of host genes were tested (Thoms et al., 383

2020b). As an unbiased interrogation of global cellular pathways affected by Nsp1, our 384

transcriptome profiling data and gene set enrichment analysis revealed strong 385

signatures of transcriptomic changes in broad ranges of host genes with several major 386

clusters, providing a comprehensive understanding of the impacts of one of the most 387

potent pathogenicity protein factors of SARS-CoV-2 in human cells of lung origin. 388

Our structure of the SARS-CoV-2 Nsp1 protein bound to the 40S ribosomal 389

subunit establishes a mechanistic basis of the cellular effects of Nsp1, revealing a 390

multifaceted mechanism of inhibition of the host protein synthesis at the initiation stage 391

by the virus. Nsp1 plugs the mRNA channel entry, which physically blocks access to the 392

channel by any mRNA (Figure 4B). Moreover, Nsp1 locks the head domain of the 40S 393

subunit in the closed position, characterized by the closed conformation of the “mRNA 394

entry channel latch” that clams around incoming mRNA (Hinnebusch, 2017b; Lomakin 395

and Steitz, 2013; Passmore et al., 2007). The latch is supposed to be closed during the 396

scanning of the mRNA, keeping mRNA locked in the binding cleft and increasing 397

processivity of the scanning, whereas the open conformation of the latch would facilitate 398

the initial attachment of the 43S PIC to the mRNA (Lomakin and Steitz, 2013). 399

Therefore, when Nsp1 keeps the latch closed it makes impossible for the host mRNA to 400

be loaded. In addition, we showed that Nsp1 competes with eIF3j for the binding to the 401

40S subunit (Figure 4H). This allows us to propose that Nsp1 weakens the binding of 402

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the eIF3 to the 40S subunit by disrupting uS3-eIF3j interaction. Recently, several 403

structures of Nsp1 bound ribosomal complexes were reported, including binary (Nsp1-404

40S), with ribosome biogenesis factor TSR1, and with eIF3-containing PICs (Schubert 405

et al., 2020; Thoms et al., 2020a). None of these structures, however, captured the 406

mRNA, which likely is flexible or dissociates from the PIC because of the lack of the 407

mRNA-eIF4F interaction. Using CrPV IRES RNA we were able to visualize the RNA 408

bound to Nsp1-40S complex and show that Nsp1 does not inhibit mRNA binding to the 409

ribosome, instead it prevents physiological conformation of the 48S PIC by restricting 410

the ribosome head domain rotation. 411

Our results explain how Nsp1 inhibits protein synthesis; however, how SARS-412

CoV-2 escapes this inhibition and initiate translation of its own RNA still remains 413

unanswered. The 5’-UTR of SARS-CoV is essential for escaping Nsp1-mediated 414

suppression of translation (Tanaka et al., 2012). Interactions involving the viral 5’ UTR 415

presumably result in the “unplugging” of Nsp1 from the 40S ribosome during the 416

initiation of viral translation. In addition, the weakening of eIF3 binding to the 40S 417

subunit is beneficial for translation initiation of some viruses. The hepatitis C virus (HCV) 418

IRES displaces eIF3 from the interface of the 40S subunit to load its RNA in the mRNA 419

binding channel (Hashem et al., 2013; Niepmann and Gerresheim, 2020). HCV IRES 420

interacts with eIF3a, eIF3c and other core subunits of eIF3 to promote formation of the 421

viral 48S PIC (Cate, 2017). The eIF3d subunit of the eIF3 complex can be cross-linked 422

to the mRNA in the exit channel of the 48S PIC, it has its own cap-binding activity which 423

can replace canonical eIF4E dependent pathway and promote translation of selected 424

cellular mRNAs (Lee et al., 2016; Pisarev et al., 2008; Walker et al., 2020). Interestingly, 425

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a recent genome-wide CRISPR screen revealed the eIF3a and eIF3d are essential for 426

SARS-CoV-2 infection (Wei et al., 2020). The requirement of the same essential 427

initiation factors suggests that it is possible that SARS-CoV-2 may use an “IRES-like” 428

mechanism involving eIF3 recruitment by 5’ UTR to overcome Nsp1 inhibition. Binding 429

of 5’ UTR may cause conformational change of the 40S head leading to the latch 430

opening, Nsp1 dissociation, viral RNA loading into mRNA binding channel and formation 431

of the functional 80S initiation complex primed for viral protein synthesis. However, the 432

detailed mechanisms of viral escape of Nsp1 inhibition must await for future 433

experimental studies. 434

435

Limitations 436

The transcriptome changes were observed in the presence of Nsp1 in the cells of 437

human lung origin. However, the role of the transcriptome changes in the loss of cell 438

viability is still not understood. To elucidate the mechanism of translation inhibition by 439

Nsp1, we determined the cryo-EM structure of rabbit 40S ribosomal subunit complex 440

with Nsp1. The atomic structure of C-Nsp1 was built into well-defined high-resolution 441

density, while only global density of the N-Nsp1 was observed. Further work is needed 442

to reveal the details and the potential functional consequence of the interaction of N-443

Nsp1 and 40S ribosome subunit. Our results suggested potential mechanisms of SARS-444

CoV-2 translation initiation, but future experiments are needed to illustrate how SARS-445

CoV-2 overcomes the Nsp1 inhibition and starts the translation of its own genome. 446

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Acknowledgements 447

We thank the Yale cryo-EM facilities for assistance with data collection. We thank the 448

Xiong lab and Chen lab members for discussions. We thank Drs. Krogan and Gordon 449

for generously providing the initial cDNA templates of SARS-CoV-2 ORFs. We thank 450

Xiaoyun Dai, Lupeng Ye, Paul Clark and several others for sharing various plasmids 451

and reagents. We thank the Yale Center for Genome Analysis and Center for Research 452

Computing for providing High-throughput sequencing and computing assistance and 453

resources. This work was supported by Yale discretionary funds to Y.X. and S.C. The 454

lab of SC is also supported by NIH (DP2CA238295, 1R01CA231112, U54CA209992-455

8697, R33CA225498, RF1DA048811) and DoD (W81XWH2010072). 456

457

Author contributions 458

S.Y., L.P., S.C., I.B.L. and Y.X. initiated the project and designed the experiments. S.Y. 459

I.B.L, Q.S. and Y.H. produced proteins and 40S ribosomal subunit. S.Y. and S.D. 460

performed binding assays. S.Y. prepared the cryo-EM samples. Y.H. and S.W. carried 461

out cryo-EM data collection. S.Y. and Y.X. did cryo-EM data processing. S.Y., I.B.L., 462

S.D. and Y.X. analyzed cryo-EM structure. L.P. and M.B.D. performed cellar assays. 463

L.P. and J.J.P. performed and processed mRNA-seq. S.Y., L.P., S.C., I.B.L. and Y.X. 464

prepared the manuscript. S.C., I.B.L. and Y.X. jointly supervised the work. 465

466

Declaration of interests 467

The authors declare no competing interests. 468

469

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Figures 470

471

Figure 1. SARS-CoV-2 ORF mini-screen identified Nsp1 as a key viral protein with 472

host cell viability effect. 473

(A) Schematics of viral protein coding frames along SARS-CoV-2 genome. Colored 474

ORFs indicate the ones used in this study, while two ORFs in grey are not (Nsp3 475

and Nsp16). 476

(B) Schematics of molecular and cellular experiments of viral proteins. 477

(C) Scatter plot of SARS-CoV-2 ORF mini-screen for host viability effect in H1299 478

cells, at 48 and 72 hours post ORF introduction. Each dot represents the mean 479

normalized relative viability of host cells transfected with a viral protein encoding 480

ORF. Dash line error bars indicate standard deviations. (n = 3 replicates). Pink 481

color indicates hits with p < 0.05 (one-way ANOVA, with multiple group 482

comparison). 483

(D) Bar plot of firefly luciferase reporter measurement of viability effects of SARS-484

CoV-2 ORFs in H1299-PL cells, at 24, 48 and 72 hours post ORF introduction (n 485

= 3 replicates). 486

(E) Bar plot of firefly luciferase reporter measurement of viability effects of Nsp1 and 487

three Nsp1 mutants (truncation, mut3: R124S/K125E and mut4: N128S/K129E) 488

in H1299-PL cells, at 24, 48 and 72 hours post ORF introduction (left, middle and 489

right panels, respectively) (n = 3 replicates). 490

(F) Flow cytometry plots of apoptosis analysis of Nsp1 and loss-of-function 491

truncation mutant in H1299-PL cells, at 48 hours post ORF introduction. 492

Percentage of apoptotic cells was gated as cleaved Caspase 3 positive cells. 493

(G) Quantification of flow-based apoptosis analysis of Nsp1 and loss-of-function 494

truncation mutant in H1299-PL cells, at 48 hours post ORF introduction. 495

For all bar plots in this figure: Bar height represents mean value and error bars 496

indicate standard error of the mean (sem). (n = 3 replicates for each group). 497

Statistical significance was accessed by ordinary one-way ANOVA, with multiple 498

group comparisons where each group was compared to empty vector control, with p-499

values subjected to multiple-testing correction by FDR method. (ns, not significant; * 500

p < 0.05; ** p < 0.01; *** p < 0.001; **** p < 0.0001). 501

See also Figure S1. 502

503

504

Figure 2. Transcriptome profiling of H1299 cells introduced with NSP1 and NSP1 505

truncation mutant by RNA-seq. 506

(A) Quantitative PCR (qPCR) confirmation of NSP1 overexpression, at 24 and 48 507

hours post electroporation. (n = 3 replicates). 508

(B) Principle component analysis (PCA) plot of the entire mRNA-seq dataset, 509

showing separation between Nsp1, Vector control and Nsp1 truncation mutant 510

groups, all electroporated into H1299-PL cells and harvested 24 hours post 511

electroporation. RNA samples were collected as quadruplicates (n = 4 each 512

group). 513

(C) Volcano plot of differential expression between of Nsp1 vs Vector Control 514

electroporated cells. Top differentially expressed genes (FDR adjusted q value < 515

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1e-100) are shown with gene names. Upregulated genes are shown in orange. 516

Downregulated genes are shown in blue. 517

(D) Bar plot of top enriched pathway analysis by DAVID Biological Processes (BP). 518

Nsp1 vs Vector control (top), or Nsp1 vs Nsp1 mutant (bottom), highly 519

downregulated (left) and upregulated (right) genes are shown (q < 1e-30). 520

See also Figure S2 521

522

523

Figure 3. Highly differentially expressed genes between Nsp1, Vector control and 524

Nsp1 mutant group in the context of top major enriched pathways. 525

(A) Gene set enrichment plots of representative enriched pathways by GSEA. 526

(B-E) Heatmap of Nsp1 highly repressed genes (q < 1e-30) in rRNA processing and 527

translation (B), mitochondria function (C), cell cycle (D), MHC-I antigen presentation 528

processes (E). 529

(F-G) Heatmap of Nsp1 highly induced genes (q < 1e-30) in polII related 530

transcription regulation processes (F) and the MAPK/ERK pathway (G). 531

See also Figure S2 532

533

534

Figure 4. cryo-EM structure of the Nsp1-40S ribosome complex. 535

(A) Overall density of the Nsp1-40S ribosome complex with Nsp1 (green) and 40S. 536

ribosome (gray). Inset shows C-Nsp1 with corresponding density with clear 537

sidechain features. C-Nsp1 α-helices (α1, aa 154-160; α2, aa 166-179) are 538

labeled. 539

(B) Cross section of the C-Nsp1 (green) within the mRNA entry channel. 40S. 540

ribosome is shown in surface and C-Nsp1 is displayed in cartoon. 541

(C) Overall density of Nsp1-40S ribosome complex at a lower contour level. Insets. 542

shows the extra globular density with SARS-CoV Nsp1 N-terminal domain 543

(PDB:2HSX, green) fitted. Ribosomal protein uS3 (magenta) and rRNA h16 544

(orange) are shown in cartoon. 545

(D) Overall structure of the C-Nsp1-40S ribosome complex, with C-Nsp1 (green. 546

surface) and the surrounding protein uS3 (magenta sphere representation), uS5 547

(cyan) and rRNA h18 (orange) highlighted. The inset shows zoomed-in view of 548

C-Nsp1 in cartoon, with the surrounding 40S components in cartoon and surface 549

to illustrate the mRNA entry channel. 550

(E) Molecular interactions between C-Nsp1 and 40S ribosome components, 551

including uS3, h18, uS5. Proteins and rRNA are in the same color as in (D) and 552

shown in cartoon, with binding pocket and hydrophobic interface depicted in 553

surface. The interacting residues are shown in sticks. 554

(F) The conformation of the 40S ribosome in the Nsp1-40S complex is similar to the. 555

close form in the 48S PIC. Q179 of uS3 (magenta cartoon) is displayed as a 556

sphere. h18 is in cartoon and colored dark yellow (48S closed conformation), 557

orange (Nsp1-40S ribosome complex) and dark green (48S open conformation), 558

with distances to Q179 indicated by the dashes. 559

(G) The N-terminal domain of Nsp1 covers uS3 surface on the solvent side. The 560

cryo-EM density in this region is shown in blue surface with SARS-CoV Nsp1 561

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N-terminal domain (PDB:2HSX) fitted. uS3 (magenta) is depicted cartoon. The 562

GEKG loop (dark purple) is shown in sphere representation. The putative 563

location of eIF3j is marked in red. 564

(H) SDS-PAGE analysis of Nsp1 and eIF3j competition at different concentration 565

ratios (indicated in the top table). 566

See also Figures S3, S4 and S5. 567

568

569

Figure 5. Nsp1 prevents physiological conformation of the 48S PIC. 570

(A) Overall structure of the Nsp1-40S-CrPV IRES complex. Nsp1 (green) and 571

IRES. (yellow) are presented in surface. The ribosome proteins (slate) and 572

rRNA (orange) are shown in cartoon. The right insets display the conformation 573

change in the Nsp1-binding region (cartoon representation) with or without the 574

IRES. 575

(B) The previously reported model of CrPV IRES (PDB: 5IT9, orange cartoon) fitted 576

to 40S ribosome in the present of Nsp1 (green cartoon). 40S ribosome (slate) 577

and the currently observed IRES (yellow) are presented in surface. 578

(C) C-Nsp1 restricts the 40S ribosome head rotation. Superposition of the Nsp1-579

40S, Nsp1-40S-CrPV IRES and IRES-40S (PDB:5IT9) complexes is shown is 580

cartoon. Zoomed view displays the head rotations represented by selected 581

rRNA regions. C-Nsp1 (green) is displayed in surface. 582

See also Figure S6. 583

584

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Table 1. Cryo-EM data collection, refinement and validation statistics, see also 585

Figure S3 and S6. 586

587

Nsp1-40S ribosome (EMDB-22432) (PDB 7JQB)

Nsp1-40S-CrPV IRES (EMDB-22433) (PDB 7JQC)

Data collection and processing Magnification 81,000 81,000 Voltage (kV) 300 300 Electron exposure (e–/Å2) 50 50 Defocus range (μm) 0.5-2.0 0.5-2.0 Pixel size (Å) 1.068 1.068 Symmetry imposed C1 C1 Initial particle images (no.) 668,695 60,690 Final particle images (no.) 353,927 48,689 Map resolution (Å) 2.7 3.3

FSC threshold. 0.143 0.143 Map resolution range (Å) 2.5-4.5 3.0-5.0 Refinement Initial model used (PDB code) 4KZX 4KZX Model resolution (Å) FSC threshold

2.7 0.143

3.3 0.143

Model resolution range (Å) Map sharpening B factor (Å2) 88 23 Model composition Non-hydrogen atoms Protein residues Ligands (nucleotide)

74,976 4,859 1,697

77,833 4,837 1,840

B factors (Å2) Protein Ligand (nucleotide)

140 150

140 167

R.m.s. deviations Bond lengths (Å) Bond angles (°)

0.007 0.8

0.006 0.9

Validation MolProbity score Clashscore Poor rotamers (%)

1.8 6.4 0.4

1.9 7.9 0.5

Ramachandran plot Favored (%) Allowed (%) Disallowed (%)

93.03 6.91 0.06

92.28 7.55 0.17

588 589

590

591

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STAR Methods. 592

593

RESOURCE AVAILABILITY 594

Lead Contact 595

Further information and requests for resources and reagents should be directed to and 596

will be fulfilled by the Lead Contact, Yong Xiong ([email protected]). 597

Material Availability 598

All unique/stable reagents generated in this study are available from the Lead Contact. 599

Data and Code Availability 600

All data generated or analyzed during this study are included in this article and its 601

supplementary information files. Specifically, source data and statistics for non-high-602

throughput experiments are provided in a supplementary table excel file (Table S2). 603

High-throughput experiment data are provided as processed quantifications in 604

Supplemental Datasets (Table S3 and S4). Genomic sequencing raw data are 605

deposited to NIH Sequence Read Archive (SRA) and/or Gene Expression Omnibus 606

(GEO) and the accession code is PRJNA667046. Constructs are available at either 607

through a public repository or via requests to the corresponding authors. Original cell 608

lines are available at commercial sources listed in supplementary information files. 609

Genetically modified cell lines are available via the authors’ laboratories. Codes that 610

support the findings of this research are being deposited to a public repository such as 611

GitHub, and are available from the corresponding authors upon reasonable request. 612

The cryo-EM maps of the Nsp1-40S ribosome complex and the Nsp1-40S-CrPV 613

IRES ribosome complex have been deposited in the Electron Microscopy Data Bank as 614

EMD-22432 and EMD-22433, respectively. The corresponding structure models are in 615

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the Protein Data Bank with accession code 7JQB, 7JQC. Additional Supplemental Items 616

are available from Mendeley Data at http://dx.doi.org/10.17632/642gjvx74d.1. 617

618

EXPERIMENTAL MODEL AND SUBJECT DETAILS 619

Mammalian cells 620

H1299, H1299-PL, Vero E6, Vero E6-PL cell lines were used in the cell viability assay 621

and the mRNA sequencing. 622

E. coli 623

E. coli BL21(DE3) was used for the expression of recombinant Nsp1 and eIF3j. 624

625

METHOD DETAILS 626

SARS-CoV-2 plasmid cloning 627

The initial cDNA templates of SARS-CoV-2 ORF gene containing plasmids were 628

provided by Dr. Krogan as a gift (Gordon et al., 2020), where the ORFs were primarily 629

cloned into lentiviral expression vector. A non-viral expression vector, pVPSB empty, 630

where ORFs were driven by a constitutive EFS promoter and terminated by a short poly 631

A, was constructed by cloning gBlock fragments (IDT) into pcDNA3.1 vector (Addgene, 632

#52535) by the Gibson assembly (NEB). All ORFs gene encoding fragments were PCR 633

amplified from the lentiviral vectors with ORF-specific forward primers and common 634

reverse primer that containing overlaps that corresponded to flanking sequences of the 635

and KpnI and XhoI restriction sites in the pVPSB empty vector. The primer lists were 636

provided in Table S1. ORFs PCR amplified fragments were gel-purified and cloned into 637

restriction enzyme digested backbone by the Gibson assembly (NEB). A lentiviral vector 638

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constitutively expressing a Firefly Luciferase and a puromycin mammalian selection 639

marker (Lenti-Fluc-Puro) was generated by standard molecular cloning. All plasmids 640

were sequenced and harvested by Maxiprep for following assay. 641

Nsp1 mutant ORF construction 642

Truncation mutant Nsp1 has triple stop codons introduced after residues 12 (N terminal 643

mutant). Nsp1 mutant3 has R124 and K125 replaced with S124 and E125 644

(R124S/K125E). Nsp1 mutant4 has N128 and K129 were converted to S128 and E129 645

(N128S/K129E). IDT gBlocks were ordered for truncated Nsp1 and different Nsp1 646

mutants with 19~23 bp overlaps that corresponded to flanking sequences of the and 647

AgeI and BstXI restriction sites in the pVPSBA01-Nsp1 plasmid. pVPSBA01-Nsp1 648

plasmid were digested and gel purified, and gBlocks were cloned using the Gibson 649

assembly (NEB). 650

Generation of stable cell lines 651

Lentivirus was produced by transfection of co-transgene plasmid (Lenti-Fluc-Puro) and 652

packaging plasmids (psPAX2, pMD2.G) into HEK293FT cells, followed by supernatant 653

harvesting, filtering and concentration with Amicon filters (Sigma). H1299 and Vero E6 654

cells were infected with Lenti-Fluc-Puro lentivirus. After 24 h of virus transduction, cells 655

were selected with 10 µg/mL puromycin, until all cells died in the control group. Luc 656

expressing H1299 and Vero E6 that with puromycin resistance cell lines were obtained 657

and named as H1299-PL and Vero E6-PL (Vero E6-PL for short) respectively. 658

Mammalian cell culture 659

H1299, H1299-PL, Vero E6, Vero E6-PL cell lines were cultured in Dulbecco’s 660

modified Eagle’s medium (DMEM; Thermo fisher) supplemented with 10% Fetal 661

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bovine serum (FBS, Hyclone),1% penicillin-streptomycin (Gibco), named as D10 662

medium. Cells were typically passaged every 1-2 days at a split ratio of 1:2 or 1:4 when 663

the confluency reached at 80%. 664

SARS-CoV-2 ORF mini-screen for cell viability 665

H1299 cells were plated in white opaque walled microwell assay plates, 25,000 cells per 666

96 well. SARS-CoV-2 ORF plasmids, 1 µg of each, were parallelly transfected with 1 µl 667

lipofectamine 2000, in triplicates. Cell viability was detected at every 24hr after 668

transfection using CellTiter-Glo® Luminescent Cell Viability Assay kit (Promega). 669

Relative viability was normalized to the mean viability of empty vector transfected 670

control group. All procedures followed the manufacturer standard protocol. Luminescent 671

signals were measured by a Plate Reader (PerkinElmer). 672

Determination of luciferase reporter cell viability 673

H1299-PL and Vero E6-PL cells were plated in white opaque walled microwell assay 674

plates, 25,000 cells per well in a 96 well. SARS-CoV-2 ORF plasmids, 1 µg of each, 675

were parallelly transfected with 1ul lipofectamine 2000. Cell viability was measured 676

every 24 hr after plasmid transfection by adding 150 µg / ml D-Luciferin (PerkinElmer) 677

using a multi-channel pipette. Luciferase intensity was measured by a Plate Reader 678

(PerkinElmer). 679

Electroporation with 4D nucleofection 680

Cells were trypsinized and collected, 1e6 cells were resuspended in SF cell line 681

NucleofectorTM solution with 3 µg plasmid DNA. Cells were transferred into 100 µl 682

NucleocuvetteTM Vessel and NCI-H1299 [H1299] cell specific protocol were utilized 683

according to the manufacturer’s protocol (4D-NucleofectorTM X Unit, Lonza). After the 684

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pulse application, 100 µl prewarmed D10 medium was added to the electroporated cells 685

in the cuvette. Cells were gently resuspended in the cuvette and transferred into 6 well 686

plate, cultured in incubator. Cells were collected at 24 or 48 hours later for 687

flowcytometry assay and RNA extraction. 688

Apoptosis flow cytometry assay 689

Flow cytometry was performed using standard immunology protocols. Briefly, 690

experimental and control cells were electroporated with respective plasmids. After a 691

defined time point, cells were collected, fixed and permeabilized using 692

Fixation/Permeablization Solution kit (BD). Then antigen-specific antibodies with 693

specific dilutions were added into cells and incubated for 30 min on ice. Cells were 694

washed with cold MACS buffer for 3 times before analyzed on a BD FACSAria 695

cytometer. Antibody used: anti-cleaved Caspase-3(Asp175) (Sigma, 9669s, 1:200). 696

Gene expression analysis by mRNA sequencing (mRNA-seq, RNA-seq) 697

For H1299-PL cells electroporated with Nsp1 or Nsp1 mutant, mRNA-seq libraries were 698

prepared following next-generation sequencing (NGS) protocols. Briefly, 1e6 H1299 699

cells were electroporated with 3 µg Nsp1, mutant Nsp1, and relative control plasmids. 700

Electroporation was done in with quadruplicates for each group. Cells were collected 701

24hr post electroporation. Total mRNA was extracted with RNasy Plus Mini Kit (Qiagen). 702

1µg total mRNA each sample was used for the RNA-seq library preparations. A 703

NEBNext® Ultra™ RNA Library Prep Kit for Illumina was employed to perform RNA-seq 704

library preparation and samples were multiplexed using barcoded primers provided by 705

NEBNext® Multiplex Oligos for Illumina® (Index Primers Set 1). All procedures follow 706

the manufacturer standard protocol. Libraries were sequenced with Novaseq system 707

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(Illumina). 708

mRNA-seq data processing, differential expression analysis and pathway 709

analysis 710

The mRNA data processing, transcript quantification, differential expression, and 711

pathway analysis were performed using custom computational programs. In brief, Fastq 712

files from mRNA-seq were used analyzed using the Kallisto quant algorithm for 713

transcript quantification (Bray et al., 2016). Differential expression analysis was 714

performed using Sleuth (Pimentel et al., 2017). Z-scores for time course heatmap were 715

calculated by log2-normalizion of gene counts following by scaling by genes. 716

Visualizations of differentially expressed genes such as volcano plots and heatmaps 717

were generated using standard R packages. Differentially upregulated and 718

downregulated genes were subjected to pathway analysis by DAVID (Huang et al., 719

2007) and/or GSEA (Subramanian et al., 2005). Processed mRNA-seq data, differential 720

expression analysis and pathway analysis results are provided in (Table S3 and S4). 721

RT-qPCR 722

Total RNA was extracted from cells using RNasy Plus Mini Kit (Qiagen). Total mRNA 723

was reverse transcribed into cDNA by M-MLV Reverse Transcriptase (Sigma). Samples 724

were collected in triplicates. Gene expression was quantified using Taqman Fast 725

Universal PCR Master Mix (Thermo Fisher) and Taqman probes (Invitrogen). NSP1 726

probe was generated with custom designed according to the Nsp1 DNA sequence in the 727

SARS-CoV-2 genome annotation (2019-nCoV/USA-WA1/2020, accession MN985325). 728

RNA expression level was normalized to ACTB (human). Relative mRNA expression 729

was determined via the ∆∆ Ct method. 730

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Ribosome and CrPV IRES purification 731

40S ribosomal subunits were purified from the rabbit reticulocyte lysate (Green 732

Hectares, USA) as described previously (Lomakin and Steitz, 2013). The gene for wild-733

type CrPV IRES (nucleotides 6028-6240) was chemically synthesized and cloned in the 734

pBluescript SK vector flanked at the 5’-end by a T7 promoter sequence and an EcoRI 735

cleavage site at the 3’-end. Standard in vitro transcription protocol was used for IRES 736

RNA synthesis and purification (MEGAscript™ T7 Transcription Kit, Ambion, USA). 737

Protein construction, expression and purification 738

Full-length SARS-CoV-2 Nsp1 was cloned into pMAT-9s vector and pET-Duet vector for 739

expression of MBP-tagged and 6×his tagged proteins, respectively. The Escherichia coli 740

BL21 (DE3) cells were used for protein expressions, which were induced by 0.5 mM 741

isopropyl β-D-1-thiogalactopyranoside (IPTG) at 16 °C for 16 hours in Terrific Broth. 742

Cells were harvested and lysed using a microfluidizer. The lysate was clarified by 743

centrifugation and then applied to a Ni-NTA (Qiagen) column. Anion exchange (HiTrap 744

Q HP, GE healthcare) chromatography was performed in a buffer of 50 mM Tris, pH 8.0 745

with a NaCl concentration gradient from 50 mM to 1M. Subsequent size exclusion 746

chromatography (HiLoad Superdex 75, GE healthcare) was performed in a buffer of 50 747

mM Tris, 150 mM NaCl, pH 8.0. Purity of the proteins was analyzed by SDS-PAGE after 748

each step. Full length eIF3j was expressed in Escherichia coli BL21 and purified with a 749

similar method. 750

Filter binding assays 751

Rabbit 40S ribosome and binding partners (proteins or CrPV IRES RNA) were 752

incubated together for 20 min at 37 °C in a total volume of 20 µl in 1× 48S buffer (20 753

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mM HEPES(KOH) pH 7.5, 100 mM KCl, 2.5 mM MgAc, 1 mM DTT, 250 µM Spermidine 754

3HCl). Reaction mixtures were incubated for another 20 min at room temperature 755

before diluting to 100 µl with H100 buffer (10 mM HEPES(KOH) pH 7.0, 100 mM KCl, 5 756

mM MgAc, 2 mM DTT). Diluted reaction mixtures were filtered through 100 kDa filter 757

(Thermo Scientific) in 10,000g for 5 min. The flow through was collected. 200 µl H100 758

buffer was used for washing the unbound proteins or RNA for 4 times before analyzing 759

by SDS-PAGE or RNA gel. 760

The concentration for the 40S ribosome for the filter binding assay is 1.5 µM and 761

the Nsp1 concentration is 15 µM (ratio of 1:10). In the Nsp1 and eIF3j competition 762

assays, the concentrations of eIF3j are 7.5 µM, 15 µM and 30 µM corresponding to 763

ratios of 1:5, 1:10 and 1:20. The concentration of the CrPV IRES is 7.5 µM in the Nsp1-764

IRES binding assay (ratio of 1:5). 765

Cryo-EM sample preparation, data collection and processing 766

40S ribosome and Nsp1, with or without the CrPV IRES RNA were mixed and incubated 767

at 37 °C for 20 mins to form a stable complex. The complex (4 µl) was applied to a C-768

Flat 2/1 3C copper grid (Electron Microscopy Sciences) pretreated by glow-discharging 769

at 8 mA for 20 seconds. The grid was blotted at 20 °C with 100% humidity and plunge-770

frozen in liquid ethane using FEI Vitrobot Mark IV (Thermo Fisher). The grids were 771

stored in liquid nitrogen before data collection. 772

Images were acquired on a FEI Titan Krios electron microscope (Thermo Fisher) 773

equipped with a post-GIF Gatan K3 direct detector in super-resolution mode, at a 774

nominal calibrated magnification of 81,000× with the physical pixel size corresponding 775

to 1.068Å. Automated data collection was performed using SerialEM (Mastronarde, 776

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34

2005). 777

A total of 4,700 movie series were collected for the Nsp1-40S ribosome complex. 778

300 movies series were collected for the Nsp1-40S-CrPV IRES complex. For the Nsp1-779

40S ribosome complex, a defocus range of 0.5 µm to 2 µm was used. Data were 780

collected with a dose of 15.9 electrons per pixel per second. Images were recorded over 781

a 3.6s exposure with 0.1s for each frame to give a total dose of 50 electrons per Å2. 782

Similar conditions were used for the Nsp1-40S-CrPV IRES complex. 783

The same data processing procedures were carried out for both the two 784

complexes using standard pipelines in cryoSPARC(Punjani et al., 2017). The final 785

average resolution is 2.7 Å for the Nsp1-40S ribosome complex and 3.3 Å for the Nsp1-786

40S-CrPV IRES complex (FSC=0.143). Local refinement was carried out for the head 787

domain of the 40S, which significantly increased the quality of the reconstruction for this 788

domain (Figure S3D). 789

Model building and refinement 790

The structure of the rabbit 40S ribosome was extracted from PDB: 4KZX (Lomakin and 791

Steitz, 2013) and 6SGC (Chandrasekaran et al., 2019). The model of Nsp1 C-terminal 792

domain was manually built in COOT (Emsley et al., 2010). The CrPV IRES structure 793

was extracted form PDB:5IT9 and refined (Murray et al., 2016). The structures of Nsp1-794

40S ribosome complex and Nsp1-IRES-40S ribosome complex were refined with 795

phenix.real_space_refine module in PHENIX (Adams et al., 2010). All structural figures 796

were generated using PyMol (Schrodinger, 2015) and Chimera (Pettersen et al., 2004). 797

798

QUANTIFICATION AND STATISTICAL ANALYSIS 799

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Sample size determination 800

Sample size was determined according to the lab's prior work or similar approaches in 801

the field. 802

Replication 803

All experiments were done with at least three biological replicates. Experimental 804

replications were indicated in detail in methods section and in each figure panel's 805

legend. 806

Standard statistical analysis 807

All statistical methods are described in figure legends and/or supplementary Excel 808

tables. The P values and statistical significance were estimated for all analyses. For 809

example, the unpaired, two-sided, T test was used to compare two groups. One-way 810

ANOVA along with multiple comparisons test, was used to compare multiple groups. 811

Multiple-testing correction was done using false discovery rate (FDR) method. Different 812

levels of statistical significance were accessed based on specific p values and type I 813

error cutoffs (0.05, 0.01, 0.001, 0.0001). Data analysis was performed using GraphPad 814

Prism v.8. and/or RStudio. 815

816

List of Supplemental Tables (provided as excel files) 817

Table S1. Oligo sequences used in this study, Related to Figure 1. 818

819

Table S2. Source data and summary statistics of cellular viability effect by 820

introduction of SARS-CoV-2 viral proteins and mutants, Related to Figure1. 821

822

Table S3. Processed Nsp1 mRNA-seq dataset and differential expression 823

analysis, Related to Figure 2. 824

Sup table 3.1 TPM table of Nsp1 mRNA-seq dataset 825

Sup table 3.2 Differential expression Nsp1 vs Vector Control 826

Sup table 3.3 Differential expression Nsp1 Mutant vs Vector Control 827

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Sup table 3.4 Differential expression Nsp1 vs Nsp1 Mutant 828

829

Table S4. DAVID pathway analysis of Nsp1 differentially expressed gene sets, 830

Related to Figure 2. 831

Sup table 4.1 Functional clustering of Nsp1 vs Vector Control highly 832

downregulated genes (q < 1e-30) 833

Sup table 4.2 Functional clustering of Nsp1 vs Nsp1 Mutant highly 834

downregulated genes (q < 1e-30) 835

Sup table 4.3 Functional clustering of Nsp1 vs Vector Control highly upregulated 836

genes (q < 1e-30) 837

Sup table 4.4 Functional clustering of Nsp1 vs Nsp1 Mutant highly upregulated 838

genes (q < 1e-30) 839

Sup table 4.5 Biological processes enrichment of Nsp1 vs Vector Control highly 840

downregulated genes (q < 1e-30) 841

Sup table 4.6 Biological processes enrichment of Nsp1 vs Nsp1 Mutant highly 842

downregulated genes (q < 1e-30) 843

Sup table 4.7 Biological processes enrichment of Nsp1 vs Vector Control highly 844

upregulated genes (q < 1e-30) 845

Sup table 4.8 Biological processes enrichment of Nsp1 vs Nsp1 Mutant highly 846

upregulated genes (q < 1e-30) 847

Sup table 4.9 Gene list of Nsp1 vs Vector Control highly downregulated genes (q 848

< 1e-30) 849

Sup table 4.10 Gene list enrichment of Nsp1 vs Nsp1 Mutant highly 850

downregulated genes (q < 1e-30) 851

Sup table 4.11 Gene list enrichment of Nsp1 vs Vector Control highly upregulated 852

genes (q < 1e-30) 853

Sup table 4.12 Gene list enrichment of Nsp1 vs Nsp1 Mutant highly upregulated 854

genes (q < 1e-30) 855

Sup table 4.13 Gene list of Nsp1 vs Vector Control all downregulated genes (q < 856

0.01) 857

Sup table 4.14 Gene list of Nsp1 vs Vector Control all upregulated genes (q < 858

0.01) 859

860

861

References: 862

Adams, P.D., Afonine, P.V., Bunkoczi, G., Chen, V.B., Davis, I.W., Echols, N., Headd, J.J., Hung, L.W., Kapral, G.J., 863 Grosse-Kunstleve, R.W., et al. (2010). PHENIX: a comprehensive Python-based system for macromolecular structure 864 solution. Acta Crystallogr D Biol Crystallogr 66, 213-221. 865 Almeida, M.S., Johnson, M.A., Herrmann, T., Geralt, M., and Wuthrich, K. (2007). Novel beta-barrel fold in the 866 nuclear magnetic resonance structure of the replicase nonstructural protein 1 from the severe acute respiratory 867 syndrome coronavirus. J Virol 81, 3151-3161. 868 Astuti, I., and Ysrafil (2020). Severe Acute Respiratory Syndrome Coronavirus 2 (SARS-CoV-2): An overview of viral 869 structure and host response. Diabetes Metab Syndr 14, 407-412. 870

Journ

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proof

Page 39: Nonstructural protein 1 of SARS-CoV-2 is a potent

37

Aylett, C.H., Boehringer, D., Erzberger, J.P., Schaefer, T., and Ban, N. (2015). Structure of a yeast 40S-eIF1-eIF1A-eIF3-871 eIF3j initiation complex. Nat Struct Mol Biol 22, 269-271. 872 Babaylova, E., Malygin, A., Gopanenko, A., Graifer, D., and Karpova, G. (2019). Tetrapeptide 60-63 of human 873 ribosomal protein uS3 is crucial for translation initiation. Bba-Gene Regul Mech 1862. 874 Blanco-Melo, D., Nilsson-Payant, B.E., Liu, W.C., Uhl, S., Hoagland, D., Moller, R., Jordan, T.X., Oishi, K., Panis, M., 875 Sachs, D., et al. (2020). Imbalanced Host Response to SARS-CoV-2 Drives Development of COVID-19. Cell 181, 1036-876 1045 e1039. 877 Bray, N.L., Pimentel, H., Melsted, P., and Pachter, L. (2016). Near-optimal probabilistic RNA-seq quantification. Nat 878 Biotechnol 34, 525-527. 879 Cate, J.H. (2017). Human eIF3: from 'blobology' to biological insight. Philos Trans R Soc Lond B Biol Sci 372. 880 Chandrasekaran, V., Juszkiewicz, S., Choi, J., Puglisi, J.D., Brown, A., Shao, S., Ramakrishnan, V., and Hegde, R.S. 881 (2019). Mechanism of ribosome stalling during translation of a poly(A) tail. Nat Struct Mol Biol 26, 1132-1140. 882 Coronaviridae Study Group of the International Committee on Taxonomy of, V. (2020). The species Severe acute 883 respiratory syndrome-related coronavirus: classifying 2019-nCoV and naming it SARS-CoV-2. Nat Microbiol 5, 536-884 544. 885 Dong, J.S., Aitken, C.E., Thakur, A., Shin, B.S., Lorsch, J.R., and Hinnebusch, A.G. (2017). Rps3/uS3 promotes mRNA 886 binding at the 40S ribosome entry channel and stabilizes preinitiation complexes at start codons. P Natl Acad Sci 887 USA 114, E2126-E2135. 888 Emsley, P., Lohkamp, B., Scott, W.G., and Cowtan, K. (2010). Features and development of Coot. Acta Crystallogr D 889 Biol Crystallogr 66, 486-501. 890 Fraser, C.S., Berry, K.E., Hershey, J.W.B., and Doudna, J.A. (2007). eIF3j is located in the decoding center of the 891 human 40S ribosomal subunit. Mol Cell 26, 811-819. 892 Fraser, C.S., Lee, J.Y., Mayeur, G.L., Bushell, M., Doudna, J.A., and Hershey, J.W.B. (2004). The j-subunit of human 893 translation initiation factor eIF3 is required for the stable binding of eIF3 and its subcomplexes to 40 S ribosomal 894 subunits in vitro. J Biol Chem 279, 8946-8956. 895 Gordon, D.E., Jang, G.M., Bouhaddou, M., Xu, J., Obernier, K., White, K.M., O'Meara, M.J., Rezelj, V.V., Guo, J.Z., 896 Swaney, D.L., et al. (2020). A SARS-CoV-2 protein interaction map reveals targets for drug repurposing. Nature 583, 897 459-468. 898 Graifer, D., Malygin, A., Zharkov, D.O., and Karpova, G. (2014). Eukaryotic ribosomal protein S3: A constituent of 899 translational machinery and an extraribosomal player in various cellular processes. Biochimie 99, 8-18. 900 Haimov, O., Sinvani, H., Martin, F., Ulitsky, I., Emmanuel, R., Tamarkin-Ben-Harush, A., Vardy, A., and Dikstein, R. 901 (2017). Efficient and Accurate Translation Initiation Directed by TISU Involves RPS3 and RPS10e Binding and 902 Differential Eukaryotic Initiation Factor 1A Regulation. Mol Cell Biol 37. 903 Hashem, Y., des Georges, A., Dhote, V., Langlois, R., Liao, H.Y., Grassucci, R.A., Pestova, T.V., Hellen, C.U., and Frank, 904 J. (2013). Hepatitis-C-virus-like internal ribosome entry sites displace eIF3 to gain access to the 40S subunit. Nature 905 503, 539-543. 906 Hershey, J.W. (2015). The role of eIF3 and its individual subunits in cancer. Biochim Biophys Acta 1849, 792-800. 907 Hertz, M.I., and Thompson, S.R. (2011). Mechanism of translation initiation by Dicistroviridae IGR IRESs. Virology 908 411, 355-361. 909 Hinnebusch, A.G. (2014). The scanning mechanism of eukaryotic translation initiation. Annu Rev Biochem 83, 779-910 812. 911 Hinnebusch, A.G. (2017a). Structural Insights into the Mechanism of Scanning and Start Codon Recognition in 912 Eukaryotic Translation Initiation. Trends Biochem Sci 42, 589-611. 913 Hinnebusch, A.G. (2017b). Structural Insights into the Mechanism of Scanning and Start Codon Recognition in 914 Eukaryotic Translation Initiation. Trends Biochem Sci 42, 589-611. 915 Hinnebusch, A.G., Ivanov, I.P., and Sonenberg, N. (2016). Translational control by 5'-untranslated regions of 916 eukaryotic mRNAs. Science 352, 1413-1416. 917 Hoffmann, M., Kleine-Weber, H., Schroeder, S., Kruger, N., Herrler, T., Erichsen, S., Schiergens, T.S., Herrler, G., Wu, 918 N.H., Nitsche, A., et al. (2020). SARS-CoV-2 Cell Entry Depends on ACE2 and TMPRSS2 and Is Blocked by a Clinically 919 Proven Protease Inhibitor. Cell 181, 271-+. 920 Huang, C., Lokugamage, K.G., Rozovics, J.M., Narayanan, K., Semler, B.L., and Makino, S. (2011). SARS coronavirus 921 nsp1 protein induces template-dependent endonucleolytic cleavage of mRNAs: viral mRNAs are resistant to nsp1-922 induced RNA cleavage. Plos Pathog 7, e1002433. 923

Journ

al Pre-

proof

Page 40: Nonstructural protein 1 of SARS-CoV-2 is a potent

38

Huang, D.W., Sherman, B.T., Tan, Q., Collins, J.R., Alvord, W.G., Roayaei, J., Stephens, R., Baseler, M.W., Lane, H.C., 924 and Lempicki, R.A. (2007). The DAVID Gene Functional Classification Tool: a novel biological module-centric 925 algorithm to functionally analyze large gene lists. Genome Biol 8, R183. 926 Jan, E., Mohr, I., and Walsh, D. (2016). A Cap-to-Tail Guide to mRNA Translation Strategies in Virus-Infected Cells. 927 Annu Rev Virol 3, 283-307. 928 Kamitani, W., Huang, C., Narayanan, K., Lokugamage, K.G., and Makino, S. (2009). A two-pronged strategy to 929 suppress host protein synthesis by SARS coronavirus Nsp1 protein. Nat Struct Mol Biol 16, 1134-U1132. 930 Kamitani, W., Narayanan, K., Huang, C., Lokugamage, K., Ikegami, T., Ito, N., Kubo, H., and Makino, S. (2006). Severe 931 acute respiratory syndrome coronavirus nsp1 protein suppresses host gene expression by promoting host mRNA 932 degradation. Proc Natl Acad Sci U S A 103, 12885-12890. 933 Kim, D., Lee, J.Y., Yang, J.S., Kim, J.W., Kim, V.N., and Chang, H. (2020). The Architecture of SARS-CoV-2 934 Transcriptome. Cell 181, 914-921 e910. 935 Lee, A.S., Kranzusch, P.J., Doudna, J.A., and Cate, J.H. (2016). eIF3d is an mRNA cap-binding protein that is required 936 for specialized translation initiation. Nature 536, 96-99. 937 Lim, Y.X., Ng, Y.L., Tam, J.P., and Liu, D.X. (2016). Human Coronaviruses: A Review of Virus-Host Interactions. 938 Diseases 4. 939 Lokugamage, K.G., Narayanan, K., Huang, C., and Makino, S. (2012). Severe Acute Respiratory Syndrome 940 Coronavirus Protein nsp1 Is a Novel Eukaryotic Translation Inhibitor That Represses Multiple Steps of Translation 941 Initiation. J Virol 86, 13598-13608. 942 Lomakin, I.B., and Steitz, T.A. (2013). The initiation of mammalian protein synthesis and mRNA scanning 943 mechanism. Nature 500, 307-311. 944 Lozano, G., and Martinez-Salas, E. (2015). Structural insights into viral IRES-dependent translation mechanisms. 945 Curr Opin Virol 12, 113-120. 946 Martinez-Salas, E., Francisco-Velilla, R., Fernandez-Chamorro, J., and Embarek, A.M. (2018). Insights into Structural 947 and Mechanistic Features of Viral IRES Elements. Front Microbiol 8. 948 Masters, P.S. (2006). The molecular biology of coronaviruses. Adv Virus Res 66, 193-292. 949 Mastronarde, D.N. (2005). Automated electron microscope tomography using robust prediction of specimen 950 movements. J Struct Biol 152, 36-51. 951 Murray, J., Savva, C.G., Shin, B.S., Dever, T.E., Ramakrishnan, V., and Fernandez, I.S. (2016). Structural 952 characterization of ribosome recruitment and translocation by type IV IRES. Elife 5. 953 Narayanan, K., Huang, C., Lokugamage, K., Kamitani, W., Ikegami, T., Tseng, C.T., and Makino, S. (2008). Severe acute 954 respiratory syndrome coronavirus nsp1 suppresses host gene expression, including that of type I interferon, in 955 infected cells. J Virol 82, 4471-4479. 956 Niepmann, M., and Gerresheim, G.K. (2020). Hepatitis C Virus Translation Regulation. Int J Mol Sci 21. 957 Passmore, L.A., Schmeing, T.M., Maag, D., Applefield, D.J., Acker, M.G., Algire, M.A., Lorsch, J.R., and Ramakrishnan, 958 V. (2007). The eukaryotic translation initiation factors eIF1 and eIF1A induce an open conformation of the 40S 959 ribosome. Mol Cell 26, 41-50. 960 Pettersen, E.F., Goddard, T.D., Huang, C.C., Couch, G.S., Greenblatt, D.M., Meng, E.C., and Ferrin, T.E. (2004). UCSF 961 Chimera--a visualization system for exploratory research and analysis. J Comput Chem 25, 1605-1612. 962 Pimentel, H., Bray, N.L., Puente, S., Melsted, P., and Pachter, L. (2017). Differential analysis of RNA-seq incorporating 963 quantification uncertainty. Nat Methods 14, 687-690. 964 Pisarev, A.V., Kolupaeva, V.G., Yusupov, M.M., Hellen, C.U., and Pestova, T.V. (2008). Ribosomal position and 965 contacts of mRNA in eukaryotic translation initiation complexes. The EMBO journal 27, 1609-1621. 966 Punjani, A., Rubinstein, J.L., Fleet, D.J., and Brubaker, M.A. (2017). cryoSPARC: algorithms for rapid unsupervised 967 cryo-EM structure determination. Nat Methods 14, 290-296. 968 Schrodinger, LLC (2015). The PyMOL Molecular Graphics System, Version 1.8. 969 Schubert, K., Karousis, E.D., Jomaa, A., Scaiola, A., Echeverria, B., Gurzeler, L.A., Leibundgut, M., Thiel, V., 970 Muhlemann, O., and Ban, N. (2020). SARS-CoV-2 Nsp1 binds the ribosomal mRNA channel to inhibit translation. 971 Nat Struct Mol Biol. 972 Sharifulin, D.E., Bartuli, Y.S., Meschaninova, M.I., Ven'yaminova, A.G., Graifer, D.M., and Karpova, G.G. (2016). 973 Exploring accessibility of structural elements of the mammalian 40S ribosomal mRNA entry channel at various 974 steps of translation initiation. Bba-Proteins Proteom 1864, 1328-1338. 975 Sharifulin, D.E., Grosheva, A.S., Bartuli, Y.S., Malygin, A.A., Meschaninova, M.I., Ven'yaminova, A.G., Stahl, J., 976

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Graifer, D.M., and Karpova, G.G. (2015). Molecular contacts of ribose-phosphate backbone of mRNA with human 977 ribosome. Bba-Gene Regul Mech 1849, 930-939. 978 Sokabe, M., and Fraser, C.S. (2014). Human eukaryotic initiation factor 2 (eIF2)-GTP-Met-tRNAi ternary complex 979 and eIF3 stabilize the 43 S preinitiation complex. J Biol Chem 289, 31827-31836. 980 Subramanian, A., Tamayo, P., Mootha, V.K., Mukherjee, S., Ebert, B.L., Gillette, M.A., Paulovich, A., Pomeroy, S.L., 981 Golub, T.R., Lander, E.S., et al. (2005). Gene set enrichment analysis: a knowledge-based approach for interpreting 982 genome-wide expression profiles. Proc Natl Acad Sci U S A 102, 15545-15550. 983 Tanaka, T., Kamitani, W., DeDiego, M.L., Enjuanes, L., and Matsuura, Y. (2012). Severe acute respiratory syndrome 984 coronavirus nsp1 facilitates efficient propagation in cells through a specific translational shutoff of host mRNA. J 985 Virol 86, 11128-11137. 986 Thoms, M., Buschauer, R., Ameismeier, M., Koepke, L., Denk, T., Hirschenberger, M., Kratzat, H., Hayn, M., Mackens-987 Kiani, T., Cheng, J., et al. (2020a). Structural basis for translational shutdown and immune evasion by the Nsp1 988 protein of SARS-CoV-2. Science 369, 1249-1255. 989 Thoms, M., Buschauer, R., Ameismeier, M., Koepke, L., Denk, T., Hirschenberger, M., Kratzat, H., Hayn, M., Mackens-990 Kiani, T., Cheng, J., et al. (2020b). Structural basis for translational shutdown and immune evasion by the Nsp1 991 protein of SARS-CoV-2. Science. 992 Tohya, Y., Narayanan, K., Kamitani, W., Huang, C., Lokugamage, K., and Makino, S. (2009). Suppression of host gene 993 expression by nsp1 proteins of group 2 bat coronaviruses. J Virol 83, 5282-5288. 994 Walker, M.J., Shortridge, M.D., Albin, D.D., Cominsky, L.Y., and Varani, G. (2020). Structure of the RNA Specialized 995 Translation Initiation Element that Recruits eIF3 to the 5'-UTR of c-Jun. J Mol Biol 432, 1841-1855. 996 Walsh, D., and Mohr, I. (2011). Viral subversion of the host protein synthesis machinery. Nat Rev Microbiol 9, 860-997 875. 998 Wathelet, M.G., Orr, M., Frieman, M.B., and Baric, R.S. (2007). Severe acute respiratory syndrome coronavirus 999 evades antiviral signaling: role of nsp1 and rational design of an attenuated strain. J Virol 81, 11620-11633. 1000 Wei, J., Alfajaro, M.M., Hanna, R.E., DeWeirdt, P.C., Strine, M.S., Lu-Culligan, W.J., Zhang, S.-M., Graziano, V.R., 1001 Schmitz, C.O., Chen, J.S., et al. (2020). Genome-wide CRISPR screen reveals host genes that regulate SARS-CoV-2 1002 infection. bioRxiv, 2020.2006.2016.155101. 1003 Wong, H.H., Kumar, P., Tay, F.P.L., Moreau, D., Liu, D.X., and Bard, F. (2015). Genome-Wide Screen Reveals Valosin-1004 Containing Protein Requirement for Coronavirus Exit from Endosomes. J Virol 89, 11116-11128. 1005 Yoshimoto, F.K. (2020). The Proteins of Severe Acute Respiratory Syndrome Coronavirus-2 (SARS CoV-2 or n-1006 COV19), the Cause of COVID-19. Protein J 39, 198-216. 1007 Zhou, P., Yang, X.L., Wang, X.G., Hu, B., Zhang, L., Zhang, W., Si, H.R., Zhu, Y., Li, B., Huang, C.L., et al. (2020). A 1008 pneumonia outbreak associated with a new coronavirus of probable bat origin. Nature 579, 270-273. 1009 Ziebuhr, J. (2005). The coronavirus replicase. Curr Top Microbiol Immunol 287, 57-94. 1010 Zust, R., Cervantes-Barragan, L., Kuri, T., Blakqori, G., Weber, F., Ludewig, B., and Thiel, V. (2007). Coronavirus non-1011 structural protein 1 is a major pathogenicity factor: Implications for the rational design of coronavirus vaccines. Plos 1012 Pathog 3, 1062-1072. 1013 1014

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Highlights

ORF screen identified Nsp1 as a major cellular pathogenicity factor of

SARS-CoV-2.

Nsp1 broadly alters the gene expression programs in human cells of lung origin.

Nsp1 inhibits translation by blocking mRNA entry channel on the 40S ribosome.

Nsp1 prevents physiological conformation of the 48S preinitiation complex (PIC).

eTOC Blurb

Yuan et al. used functional and cryo-EM studies to show that SARS-CoV-2 Nsp1

significantly reduces cell viability, induces extensive transcriptome alteration, and

blocks host mRNA access to the ribosome. These results help understand how

Nsp1 suppresses host gene expression and its broad impact as a

comorbidity-inducing factor.

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